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Original research
Choroid plexus enlargement in paediatric multiple sclerosis: clinical relevance and effect of sex
  1. Monica Margoni1,2,
  2. Mor Gueye1,
  3. Alessandro Meani1,
  4. Elisabetta Pagani1,
  5. Lucia Moiola2,
  6. Paolo Preziosa1,2,3,
  7. Massimo Filippi1,2,3,4,5,
  8. Maria A Rocca1,2,3
  1. 1Neuroimaging Research Unit, Division of Neuroscience, IRCCS Ospedale San Raffaele, Milan, Italy
  2. 2Neurology Unit, IRCCS Ospedale San Raffaele, Milan, Italy
  3. 3Vita-Salute San Raffaele University, Milan, Italy
  4. 4Neurorehabilitation Unit, IRCCS Ospedale San Raffaele, Milan, Italy
  5. 5Neurophysiology Service, IRCCS Osepdale San raffaele, Milan, Italy
  1. Correspondence to Professor Maria A Rocca, Neuroimaging Research Unit, Division of Neuroscience, IRCCS Ospedale San Raffaele, 20132 Milano, Italy; rocca.mara{at}hsr.it

Abstract

Background Choroid plexus (CP) enlargement has been suggested as a reliable marker of neuroinflammation in adult multiple sclerosis (MS). We investigated CP volume in patients with paediatric MS compared with matched healthy controls (HC), possible sex-related effect, and the associations with clinical and structural MRI variables.

Methods Brain 3.0 T dual-echo and three-dimensional (3D) T1-weighted sequences were selected retrospectively from 69 patients with paediatric MS and 23 age-matched and sex-matched HC. CP volume was manually obtained from 3D T1-weighted scans by two expert raters.

Results CP segmentation was highly reproducible (intraobserver agreement: rater I=0.963, rater II=0.958; interobserver agreement=0.968). Compared with HC, patients with paediatric MS showed higher normalised CP volume (p<0.001). Both female and male patients with paediatric MS showed higher normalised CP volume compared with sex-matched HC (women: p<0.001 and men: p=0.021), with a significant disease×sex interaction (p=0.040). In patients with MS, a higher normalised CP volume was significantly associated with higher brain lesional volume (β=0.252, p=0.017), larger lateral ventricle volume (β=0.470, false discovery rate (FDR)-p<0.001), lower normalised brain volume (β=−0.413, FDR-p=0.002) and lower normalised thalamic volume (β=0.291, FDR-p=0.046). No associations with disease duration, Expanded Disability Status Scale score, normalised cortical and white matter volumes were found (FDR-p≥0.172). A significant effect of the disease in the negative association between normalised volumes of CP and thalami was observed (FDR-p=0.046).

Conclusions CP enlargement occurs in paediatric MS, suggesting its early involvement in the pathophysiology of the disease. The higher CP volume, which is found especially in female patients, supports the hypothesis of sex-related differences occurring already in paediatric MS.

  • MULTIPLE SCLEROSIS
  • PAEDIATRIC
  • MRI

Data availability statement

Data are available upon reasonable request. The data set used and analysed during the current study are available from the corresponding author on reasonable request.

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Data availability statement

Data are available upon reasonable request. The data set used and analysed during the current study are available from the corresponding author on reasonable request.

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Footnotes

  • Twitter @monicamargoni, @paolopreziosa

  • Contributors Concept and design: MF and MAR. Acquisition, analysis or interpretation of data: MM, MG, AM, EP, LM, PP, MF and MAR. Drafting of the manuscript: MM, MG, AM, EP, LM, PP, MF and MAR. Statistical analysis: AM. MAR is responsible for the overall content as guarantor.

  • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

  • Competing interests MM reports grants and personal fees from Almirall. She was awarded a MAGNIMS-ECTRIMS fellowship in 2020. MG has nothing to disclose. AM received speakers’ honoraria from Biogen Idec. EP received speakers’ honoraria from Biogen Idec. LM received personal compensation for consulting, serving on a scientific advisory board, speaking or other activities with Sanofi-Genzyme, Novartis, Teva, Merck Serono, Biogen, Roche, and Excemed. PP received speaker honoraria from Roche, Biogen, Novartis, Merck Serono, Bristol Myers Squibb and Genzyme. He has received research support from Italian Ministry of Health and Fondazione Italiana Sclerosi Multipla. MF is Editor in-Chief of the Journal of Neurology, Associate Editor of Human Brain Mapping, Associate Editor of Radiology and Associate Editor of Neurological Sciences; received compensation for consulting services from Alexion, Almirall, Biogen, Merck, Novartis, Roche, Sanofi; speaking activities from Bayer, Biogen, Celgene, Chiesi Italia SpA, Eli Lilly, Genzyme, Janssen, Merck-Serono, Neopharmed Gentili, Novartis, Novo Nordisk, Roche, Sanofi, Takeda, and TEVA; participation in Advisory Boards for Alexion, Biogen, Bristol Myers Squibb, Merck, Novartis, Roche, Sanofi, Sanofi-Aventis, Sanofi-Genzyme, Takeda; scientific direction of educational events for Biogen, Merck, Roche, Celgene, Bristol Myers Squibb, Lilly, Novartis, Sanofi-Genzyme; he receives research support from Biogen Idec, Merck-Serono, Novartis, Roche, Italian Ministry of Health, Fondazione Italiana Sclerosi Multipla, and ARiSLA (Fondazione Italiana di Ricerca per la SLA). MAR received consulting fees from Biogen, Bristol Myers Squibb, Eli Lilly, Janssen, Roche; and speaker honoraria from Bayer, Biogen, Bristol Myers Squibb, Bromatech, Celgene, Genzyme, Merck Healthcare Germany, Merck Serono SpA, Novartis, Roche and Teva. She receives research support from the MS Society of Canada and Fondazione Italiana Sclerosi Multipla. She is the Associate Editor for Multiple Sclerosis and Related Disorders.

  • Provenance and peer review Not commissioned; externally peer reviewed.